Review
Partial duplication of the jaw: case reports and review of relevant publications

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Abstract

Craniofacial duplication is a rare congenital malformation with a wide phenotypic range. The signs and symptoms range from partial craniofacial duplication to bicephalus. We describe two cases of partial duplication of jaw: a girl with a duplication of the maxilla, and a boy with duplication of the mandible. We review the relevant publications and discuss the pathogenesis.

Introduction

Craniofacial duplication is a rare congenital malformation with a wide phenotypic range, and the incidence in newborns is about 1:2.5 million.1 The manifestations range from partial craniofacial duplication to bicephalus,2 and the most common type of partial craniofacial duplication is maxillary or mandibular duplication – or partial duplication of the jaw.3 We describe two cases: a girl with duplication of the maxilla, and a boy with duplication of the mandible. We examined the specimens histologically to try to establish the pathogenesis. We also reviewed relevant publications about different kinds of partial duplication of the jaw.

Section snippets

Case 1

Social and relevant medical and dental history: a 9-year-old girl was referred to West China Hospital of Stomatology with supernumerary teeth on her left maxilla. She was born to a mother who had had no exposure to medications, drugs, alcohol, or radiation. There was no history of family craniofacial anomalies.

Clinical findings: the patient was physically and mentally normal for her age, and maxillofacial examination showed obvious facial asymmetry. The middle third of the left side of her face

Search for relevant publications and results

We searched for relevant papers within MEDLINE (through PubMed, 1948 to September 2016). The MeSH headings and free text words were combined. They included “maxillary duplication,’’ “mandibular duplication,’’ and so on. Language was restricted to English.

The result showed that 34 papers about partial duplication of the jaw have been published, including 36 case reports.

Maxillary duplication: Table 1 is the overview of the papers about maxillary duplication. A total of 16 papers were found that

Discussion

We have described two rare clinical cases: a girl with duplication of the left maxilla, and a boy with duplication of the left mandible. There have been dozens of reports on duplication of the jaw, but we found that duplication of the mandibular condyle was rarely reported. Peacock et al reported a boy whose mandibular coronoid process was morphologically abnormal and it’s shape was similar to that of the condyle.5 Al-Ani et al described a patient with duplication of the bilateral mandibular

Conflict of interest

We have no conflicts of interest.

Ethics statement/confirmation of patients’ permission

Ethics approval was not required. Informed consent was obtained from patients, parents, or guardians.

Acknowledgements

This study is supported by Grants from the NSFC 81621062, Key Research and Development Program of Science and Technology Department of Sichuan Province2018SZ0119Sichuan Province Science and Technology Innovation Team Program2017TD0016).

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